Publications
Primary Research Articles
Lee YJ, Chang YT, Cho Y, Kowalczyk M, Dragoiescu A, Pacis A, Kailasam S, Lefebvre F, Zhang Q, Gao X, Huang WH
Molecular and developmental deficits in Smith-Magenis syndrome human stem cell-derived cortical neural models
Am J Hum Genet., 2025 Aug 26:S0002-9297(25)00310-6.
Molecular and developmental deficits in Smith-Magenis syndrome human stem cell-derived cortical neural models
Am J Hum Genet., 2025 Aug 26:S0002-9297(25)00310-6.
Kowalczyk M, Lee YJ, Huang WH
TSC2-mTORC1 axis regulates morphogenesis and neurological function of Gli1+ adult-born dentate granule cells
Mol Biol Cell, 2025 Jan 1;36(1):br1.
TSC2-mTORC1 axis regulates morphogenesis and neurological function of Gli1+ adult-born dentate granule cells
Mol Biol Cell, 2025 Jan 1;36(1):br1.
Chang YT, Lee YJ, Haque M, Chang HC, Javed S, Lin YC, Abramovitz J, Chin G, Khamis A, Raja R, Murai KK, Huang WH
Comparative analyses of the Smith-Magenis syndrome protein RAI1 in mice and common marmoset monkeys
J Comp Neurol., 2024 Jan;532(1):e25589.
Comparative analyses of the Smith-Magenis syndrome protein RAI1 in mice and common marmoset monkeys
J Comp Neurol., 2024 Jan;532(1):e25589.
Javed S, Chang YT, Cho Y, Lee YJ, Chang HC, Haque M, Lin YC, Huang WH
Smith–Magenis syndrome protein RAI1 regulates body weight homeostasis through hypothalamic BDNF-producing neurons and neurotrophin downstream signalling
Elife, 2023 Nov 13:12:RP90333
Smith–Magenis syndrome protein RAI1 regulates body weight homeostasis through hypothalamic BDNF-producing neurons and neurotrophin downstream signalling
Elife, 2023 Nov 13:12:RP90333
Chang HC, Lee YJ, Javed S, Haque M, Chang YT, Lin YC, Oram C, Huang WH
rAAV-CRISPRa therapy corrects Rai1 haploinsufficiency and rescues selective disease features in Smith-Magenis syndrome mice
J Biol Chem., 2023 Jan;299(1):102728
rAAV-CRISPRa therapy corrects Rai1 haploinsufficiency and rescues selective disease features in Smith-Magenis syndrome mice
J Biol Chem., 2023 Jan;299(1):102728
Chang YT, Kowalczyk M, Fogerson PM, Lee JY, Haque M, Adams EL, Wang DC, DeNardo LA, Tessier-Lavigne M, Huguenard JR, Luo L, Huang WH
Loss of Rai1 enhances hippocampal excitability and epileptogenesis in mouse models of Smith-Magenis syndrome
Proc Natl Acad Sci USA, 2022 Oct 25;119(43):e2210122119
Loss of Rai1 enhances hippocampal excitability and epileptogenesis in mouse models of Smith-Magenis syndrome
Proc Natl Acad Sci USA, 2022 Oct 25;119(43):e2210122119
Javed S, Lee YJ, Xu J, Huang WH
Temporal dissection of Rai1 function reveals brain-derived neurotrophic factor as a potential therapeutic target for Smith-Magenis Syndrome
Hum Mol Genet., 2021 Dec 27;31(2):275-288
Temporal dissection of Rai1 function reveals brain-derived neurotrophic factor as a potential therapeutic target for Smith-Magenis Syndrome
Hum Mol Genet., 2021 Dec 27;31(2):275-288
Huang WH, Wang DC, Allen, WE, Klope M, Hu H, Shamloo M, Luo L
Early Adolescent Rai1 Reactivation Reverses Transcriptional and Social Interaction Deficits In a Mouse Model of Smith-Magenis Syndrome
Proc Natl Acad Sci USA, 2018, Oct 16;115(42):10744-10749
Early Adolescent Rai1 Reactivation Reverses Transcriptional and Social Interaction Deficits In a Mouse Model of Smith-Magenis Syndrome
Proc Natl Acad Sci USA, 2018, Oct 16;115(42):10744-10749
Huang WH, Guenthner CJ, Xu J, Nguyen N, Wilkinson AW, Gozani O, Chang HY, Shamloo M, Luo L
Molecular and Neural Functions of Rai1, The Causal Gene for Smith-Magenis Syndrome
Neuron, 2016 Oct 19;92(2):392-406
Molecular and Neural Functions of Rai1, The Causal Gene for Smith-Magenis Syndrome
Neuron, 2016 Oct 19;92(2):392-406
Huang WH, Tupal S, Huang TW, Ward CS, Neul JL, Klisch TJ, Gray PA, Zoghbi HY
Atoh1 governs the migration of postmitotic neurons that shape respiratory effectiveness at birth and chemoresponsiveness in adulthood
Neuron, 2012 Sep 6;75(5):799-809
Atoh1 governs the migration of postmitotic neurons that shape respiratory effectiveness at birth and chemoresponsiveness in adulthood
Neuron, 2012 Sep 6;75(5):799-809
Collaborating Research Articles, Reviews, and Book Chapters
Katz N, An C, Lee YJ, Tycko J, Zhang M, Kang J, Bintu L, Bassik MC, Huang WH, Gao X
Tunable, self-contained gene dosage control via proteolytic cleavage of CRISPR-Cas systems
bioRxiv, [Preprint]. 2024 Oct 9:2024.10.09.617463.
Tunable, self-contained gene dosage control via proteolytic cleavage of CRISPR-Cas systems
bioRxiv, [Preprint]. 2024 Oct 9:2024.10.09.617463.
Huang WH
Performing Single Cell Clonal Analysis in the Mouse Brain using Mosaic Analysis with Double Markers (MADM)
Methods Mol Biol., 2022 July;2515:59-74
Performing Single Cell Clonal Analysis in the Mouse Brain using Mosaic Analysis with Double Markers (MADM)
Methods Mol Biol., 2022 July;2515:59-74
Javed S, Selliah T, Lee YJ, Huang WH
Dosage-Sensitive Genes in Autism Spectrum Disorders: From Neurobiology to Therapy
Neurosci & Biobehav Rev., 2020 Nov;118:538-567
Dosage-Sensitive Genes in Autism Spectrum Disorders: From Neurobiology to Therapy
Neurosci & Biobehav Rev., 2020 Nov;118:538-567
Luo L, Ambrozkiewicz MC, Benseler F, Chen C, Dumontier E, Falkner S, Furlanis E, Gomez AM, Hoshina N, Huang WH, Hutchison MA, Itoh-Maruoka Y, Lavery LA, Li W, Maruo T, Motohashi J, Pai ELL, Pelkey KA, Pereira A, Philips T, Sinclair JL, Stogsdill JA, Traunmuller L, Wang J, Wortel J, You W, Abumaria N, Beier KT, Brose N, Burgess HA, Cepko CL, Cloutier JF, Eroglu C, Goebbels S, Kaeser PS, Kay JN, Lu W, Luo L, Mandai K, McBain CJ, Nave KA, Prado MAM, Prado VF, Rothstein J, Rubesntein JL, Saher G, Sakimura K, Sanes JR, Scheiffele P, Takai Y, Umemori H, Verhage M, Yuzaki M, Zoghbi HY, Kawabe H, Craig AM
Optimizing Nervous System-Specific Gene Targeting with Cre Driver Lines: Prevalence of Germline Recombination and Influencing Factors
Neuron, 2020 Apr 8;106(1):37-65.e5
Optimizing Nervous System-Specific Gene Targeting with Cre Driver Lines: Prevalence of Germline Recombination and Influencing Factors
Neuron, 2020 Apr 8;106(1):37-65.e5
Yeh SY, Huang WH, Wang W, Ward CS, Chao ES, Wu Z, Tang B, Tang J, Sun JJ, van der Heijden ME, Gray PA, Xue M, Ray RS, Ren D, and Zoghbi HY
Respiratory network stability and modulatory response to substance P require Nalcn
Neuron, 2017 Apr 19;94(2):294-303.e4
Respiratory network stability and modulatory response to substance P require Nalcn
Neuron, 2017 Apr 19;94(2):294-303.e4
Tupal S, Huang WH, Picardo MC, Ling GY, Del Negro CA, Zoghbi HY, Gray PA
Atoh1-dependent rhombic lip neurons are required for temporal delay between independent respiratory oscillators in embryonic mice
Elife, 2014 May 14;3:e02265
Atoh1-dependent rhombic lip neurons are required for temporal delay between independent respiratory oscillators in embryonic mice
Elife, 2014 May 14;3:e02265